Department of Pulmonary and Critical Care Medicine, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology, Wuhan, China.
Key Laboratory of Respiratory Diseases, National Ministry of Health of the People's Republic of China and National Clinical Research Center for Respiratory Disease, Wuhan, China.
Front Endocrinol (Lausanne). 2021 Dec 23;12:770871. doi: 10.3389/fendo.2021.770871. eCollection 2021.
Woodhouse-Sakati syndrome (WSS) (OMIM#241080) is a rare multi-system autosomal recessive disease with homozygous mutation of the DCAF17 gene. The main features of WSS include diabetes, hypogonadism, alopecia, deafness, intellectual disability and progressive extrapyramidal syndrome. We identified a WSS family with a novel DCAF17 gene mutation type in China. Two unconsanguineous siblings from the Chinese Han family exhibiting signs and symptoms of Woodhouse-Sakati syndrome were presented for evaluation. Whole-exome sequencing revealed a homozygous deletion NM_025000.4:c.1488_1489delAG in the DCAF17 gene, which resulted in a frameshift mutation that led to stop codon formation. We found that the two patients exhibited low insulin and C-peptide release after glucose stimulation by insulin and C-peptide release tests. These findings indicate that the DCAF17 gene mutation may cause pancreatic β cell functional impairment and contribute to the development of diabetes.
伍德豪斯-萨卡蒂综合征(WSS)(OMIM#241080)是一种罕见的常染色体隐性遗传病,由 DCAF17 基因突变引起。WSS 的主要特征包括糖尿病、性腺功能减退、脱发、耳聋、智力障碍和进行性锥体外系综合征。我们在中国发现了一个具有新型 DCAF17 基因突变类型的 WSS 家族。这对来自中国汉族的非近亲兄弟姐妹表现出伍德豪斯-萨卡蒂综合征的迹象和症状,前来接受评估。全外显子组测序显示 DCAF17 基因中的纯合缺失 NM_025000.4:c.1488_1489delAG,导致移码突变并形成终止密码子。我们发现这两个患者在胰岛素和 C 肽释放试验中表现出葡萄糖刺激后胰岛素和 C 肽释放低。这些发现表明 DCAF17 基因突变可能导致胰岛β细胞功能障碍,导致糖尿病的发生。